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Prader-Willi syndrome imprinting centre deletion mice have impaired baseline and 5-HT2CR-mediated response inhibition.

Davies JR, Wilkinson LS, Isles AR, Humby T.

Hum Mol Genet. 2019 May 14. pii: ddz100. doi: 10.1093/hmg/ddz100. [Epub ahead of print]


Increased alternate splicing of Htr2c in a mouse model for Prader-Willi syndrome leads disruption of 5HT2C receptor mediated appetite.

Garfield AS, Davies JR, Burke LK, Furby HV, Wilkinson LS, Heisler LK, Isles AR.

Mol Brain. 2016 Dec 8;9(1):95.


Behavioural and cognitive abnormalities in an imprinting centre deletion mouse model for Prader-Willi syndrome.

Relkovic D, Doe CM, Humby T, Johnstone KA, Resnick JL, Holland AJ, Hagan JJ, Wilkinson LS, Isles AR.

Eur J Neurosci. 2010 Jan;31(1):156-64. doi: 10.1111/j.1460-9568.2009.07048.x. Epub 2009 Dec 23.


Influence of the Prader-Willi syndrome imprinting center on the DNA methylation landscape in the mouse brain.

Brant JO, Riva A, Resnick JL, Yang TP.

Epigenetics. 2014 Nov;9(11):1540-56. doi: 10.4161/15592294.2014.969667.


An unexpected function of the Prader-Willi syndrome imprinting center in maternal imprinting in mice.

Wu MY, Jiang M, Zhai X, Beaudet AL, Wu RC.

PLoS One. 2012;7(4):e34348. doi: 10.1371/journal.pone.0034348. Epub 2012 Apr 4.


Whole genome microarray analysis of gene expression in an imprinting center deletion mouse model of Prader-Willi syndrome.

Bittel DC, Kibiryeva N, McNulty SG, Driscoll DJ, Butler MG, White RA.

Am J Med Genet A. 2007 Mar 1;143A(5):422-9.


Caralluma fimbriata extract activity involves the 5-HT2c receptor in PWS Snord116 deletion mouse model.

Griggs JL, Mathai ML, Sinnayah P.

Brain Behav. 2018 Dec;8(12):e01102. doi: 10.1002/brb3.1102. Epub 2018 Oct 23.


Prader-Willi Syndrome: Obesity due to Genomic Imprinting.

Butler MG.

Curr Genomics. 2011 May;12(3):204-15. doi: 10.2174/138920211795677877.


Calorie seeking, but not hedonic response, contributes to hyperphagia in a mouse model for Prader-Willi syndrome.

Davies JR, Humby T, Dwyer DM, Garfield AS, Furby H, Wilkinson LS, Wells T, Isles AR.

Eur J Neurosci. 2015 Aug;42(4):2105-13. doi: 10.1111/ejn.12972. Epub 2015 Jun 25.


A human imprinting centre demonstrates conserved acquisition but diverged maintenance of imprinting in a mouse model for Angelman syndrome imprinting defects.

Johnstone KA, DuBose AJ, Futtner CR, Elmore MD, Brannan CI, Resnick JL.

Hum Mol Genet. 2006 Feb 1;15(3):393-404. Epub 2005 Dec 20.


Targeting the histone methyltransferase G9a activates imprinted genes and improves survival of a mouse model of Prader-Willi syndrome.

Kim Y, Lee HM, Xiong Y, Sciaky N, Hulbert SW, Cao X, Everitt JI, Jin J, Roth BL, Jiang YH.

Nat Med. 2017 Feb;23(2):213-222. doi: 10.1038/nm.4257. Epub 2016 Dec 26.


Prader-Willi syndrome and atypical submicroscopic 15q11-q13 deletions with or without imprinting defects.

Hassan M, Butler MG.

Eur J Med Genet. 2016 Nov;59(11):584-589. doi: 10.1016/j.ejmg.2016.09.017. Epub 2016 Sep 19.


Loss of the imprinted snoRNA mbii-52 leads to increased 5htr2c pre-RNA editing and altered 5HT2CR-mediated behaviour.

Doe CM, Relkovic D, Garfield AS, Dalley JW, Theobald DE, Humby T, Wilkinson LS, Isles AR.

Hum Mol Genet. 2009 Jun 15;18(12):2140-8. doi: 10.1093/hmg/ddp137. Epub 2009 Mar 20.


Deletion of the Snord116/SNORD116 Alters Sleep in Mice and Patients with Prader-Willi Syndrome.

Lassi G, Priano L, Maggi S, Garcia-Garcia C, Balzani E, El-Assawy N, Pagani M, Tinarelli F, Giardino D, Mauro A, Peters J, Gozzi A, Grugni G, Tucci V.

Sleep. 2016 Mar 1;39(3):637-44. doi: 10.5665/sleep.5542.


Paradoxical leanness in the imprinting-centre deletion mouse model for Prader-Willi syndrome.

Golding DM, Rees DJ, Davies JR, Relkovic D, Furby HV, Guschina IA, Hopkins AL, Davies JS, Resnick JL, Isles AR, Wells T.

J Endocrinol. 2017 Jan;232(1):123-135. Epub 2016 Oct 31.


Prader-Willi Critical Region, a Non-Translated, Imprinted Central Regulator of Bone Mass: Possible Role in Skeletal Abnormalities in Prader-Willi Syndrome.

Khor EC, Fanshawe B, Qi Y, Zolotukhin S, Kulkarni RN, Enriquez RF, Purtell L, Lee NJ, Wee NK, Croucher PI, Campbell L, Herzog H, Baldock PA.

PLoS One. 2016 Jan 29;11(1):e0148155. doi: 10.1371/journal.pone.0148155. eCollection 2016.


Genomic imprinting: potential function and mechanisms revealed by the Prader-Willi and Angelman syndromes.

Glenn CC, Driscoll DJ, Yang TP, Nicholls RD.

Mol Hum Reprod. 1997 Apr;3(4):321-32. Review.


Genomic analysis of the chromosome 15q11-q13 Prader-Willi syndrome region and characterization of transcripts for GOLGA8E and WHCD1L1 from the proximal breakpoint region.

Jiang YH, Wauki K, Liu Q, Bressler J, Pan Y, Kashork CD, Shaffer LG, Beaudet AL.

BMC Genomics. 2008 Jan 28;9:50. doi: 10.1186/1471-2164-9-50.

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